Testosterone‐Secreting Mature Ovarian Teratoma Causing Severe Virilization in an Adolescent

Abstract

A 15-year-old nulligravida came to our clinic with hirsutism, acne, voice changes, menometrorrhagia, clitoromegaly, and an abdominal mass. She had her menarche at 12 years of age and, since then, had had regular periods. She was never sexually active. Two years earlier, she was hospitalized for several months with seizures and a mental status change that was diagnosed as Mycoplasma encephalitis. A few months after her discharge, hirsutism and aggressive behavioral changes developed, which were attributed to her encephalitis. This was followed by development of an adult masculine voice and irregular periods. Her vocal cords were visualized and were found to be normal. The patient was referred to our clinic after she was noted to have increased abdominal girth, and a large cystic mass was palpated in the left adnexa. Case ReportSonographic and Color Doppler Characteristics yperandrogenism, associated with hirsutism in adolescent girls and premenopausal women, is a common cause of irregular menses; it affects 5% to 7% of adult women.1 The most common etiology of androgen disorders is polycystic ovary syndrome, occurring in 70% to 80% of cases. 1,2Androgen-secreting neoplasms are rare and account for less than 0.3% of patients with hirsutism.2 These neoplasms should be suspected when the onset of androgenic symptoms is rapid and sudden. We report a rare case of an adolescent girl with severe virilism and behavioral changes associated with a testosterone-secreting mature ovarian teratoma.H

Virilism and Ectopic Expression of HSD17B5 in Mature Cystic Teratoma

“…Resection of the tumor normalized serum testosterone levels, suggesting that the elevated serum testosterone level and masculinization were caused by the tumor. Previously reported mechanisms by which teratomas are known to cause masculinization include peripheral luteinization of stromal cells (Freedman andScully 1970, Lopez-Beltran et al 1997), differentiation to Leydig cells (Bulwa and Lewis 1971, Wu et al 2008, Rotenberg et al 2009), concurrent hyperthecosis (Aiman et al 1977), Brenner tumor-containing MCT (Tsujimoto et al 2011), and hilus cell hyperplasia (Rutgers and Scully 1986). None of these histological findings was detected during the Table 2.…”

“…Hyper insulin- emia reinforces testosterone action via insulin effects in the pituitary gland, liver, and ovary (Diamanti-Kandarakis and Papavassiliou 2006). Obesity or diabetes has been previously reported in cases of virilism with MCT (Aiman et al 1977, Lopez-Beltran et al 1997, Rotenberg et al 2009). However, the long-term evaluation of diabetes and insulin resistance in this case could not be performed because of the patient's circumstances.…”